Intestinal Angina in a Patient with Hypertrophic Obstructive
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Hamaoka et al. Journal of Medical Case Reports (2016) 10:271 DOI 10.1186/s13256-016-1055-8 CASE REPORT Open Access Intestinal angina in a patient with hypertrophic obstructive cardiomyopathy: a case report Takuto Hamaoka1*, Wataru Omi2, Yoshiteru Sekiguti2, Shigeo Takata2, Shuichi Kaneko1, Oto Inoue2, Shinichiro Takashima2, Hisayoshi Murai1, Soichiro Usui1, Takeshi Kato1, Hiroshi Furusho1 and Masayuki Takamura1 Abstract Background: Intestinal angina is characterized by recurrent postprandial abdominal pain and anorexia. Commonly, these symptoms are caused by severe stenosis of at least two vessels among the celiac and mesenteric arteries. However, intestinal perfusion is affected not only by the degree of arterial stenosis but also by systemic perfusion. We experienced a unique case of intestinal angina caused by relatively mild stenosis of the abdominal arteries complicated with hypertrophic obstructive cardiomyopathy. Case presentation: We report an 86-year old Japanese man with hypertrophic obstructive cardiomyopathy and advanced atrioventricular block who was diagnosed with intestinal angina. Computed tomography showed mild stenosis of the celiac artery and severe stenosis of the inferior mesenteric artery, and these lesions were relatively mild compared with other reports. A dual-chamber pacemaker with right ventricular apical pacing was implanted to improve the obstruction of the left ventricular outflow tract. After implantation, the patient’sabdominalsymptoms diminished markedly, and improvement of the left ventricular outflow tract obstruction was observed. Conclusions: Although intestinal angina is generally defined by severe stenosis of at least two vessels among the celiac and mesenteric arteries, the present case suggests that hemodynamic changes can greatly affect intestinal perfusion and induce intestinal angina in the presence of mild stenosis of the celiac and mesenteric arteries. Keywords: Intestinal angina, Hypertrophic obstructive cardiomyopathy, Advanced atrioventricular block, Case report Background tract (LVOT), resulting in an increased LVOT pressure Intestinal angina is caused by chronic intestinal ischemia gradient. On the other hand, hypertension increases the characterized by recurrent postprandial abdominal pain cardiac afterload, which inhibits the collapse of LVOT and and a decrease in body weight with anorexia [1]. The results in a decreased LVOT pressure gradient [3]. symptom is commonly manifested when at least two ves- In this report, we describe a unique case of intestinal sels of the celiac and mesenteric arteries have developed angina complicated with HOCM and abruptly worsened moderate to severe stenosis or occlusion. Hypertrophic by advanced atrioventricular (AV) block in the presence obstructive cardiomyopathy (HOCM) is associated with a of mild stenosis of the abdominal arteries. reduction in cardiac output and systemic perfusion fluctu- ated by multiple hemodynamic conditions [2] such as hypovolemia or high blood pressure. Hypovolemic condi- Case presentation tions lead to a reduction in left ventricular volume and An 86-year old Japanese man was admitted to our hospital the narrowing or collapse of the left ventricular outflow because of worsening postprandial abdominal pain, an- orexia, and general malaise. He was previously diagnosed * Correspondence: [email protected] with HOCM on cardiac magnetic resonance imaging and 1 Department of Disease Control and Homeostasis, Graduate School of echocardiography, but because he was asymptomatic, he Medical Science, Kanazawa University, 13-1 Takara-machi, Kanazawa 920-8641, Japan was not taking oral medications. He had been suffering Full list of author information is available at the end of the article from repetitive and paroxysmal upper abdominal pain for © 2016 The Author(s). Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Hamaoka et al. Journal of Medical Case Reports (2016) 10:271 Page 2 of 5 1 year. His symptoms occurred approximately 15 minutes An electrocardiogram revealed advanced AV block and after eating and usually disappeared within 1 hour and left high voltage with ST-T change; AV disturbance had were sometimes accompanied by watery diarrhea. Once not been detected previously (Fig. 3). Transthoracic echo- his abdominal attacks occurred, he had great difficulties cardiography showed significant obstruction of the LVOT with oral intake due to postprandial pain, but his symp- (pressure gradient, 35 mmHg) and mitral regurgitation toms disappeared upon fluid replacement for several days. with systolic anterior motion of the anterior mitral leaflet He presented to our department because of worsening (Fig. 4). These echocardiographic findings were similar to and more sustained symptoms within the week prior. those of echocardiography performed the previous year He was emaciated and exhausted and had lost 7 kg in on our patient. the last 1 year. A physical examination showed a low CT angiography was classified as mild to account for blood pressure (90/60 mmHg) and bradycardia (40 beats our patient’s severe postprandial pain, since our patient’s per minute). He had no signs of disorientation or paraly- superior mesenteric artery, which had the widest sis. A holosystolic ejection murmur at the fourth left intestinal perfusion area, was patent in this patient; how- sternal border was auscultated, and abdominal palpita- ever, in many previous reports concerning intestinal an- tion showed no abnormal findings. The murmur was gina, stenosis of this artery was demonstrated, and accentuated by the Valsalva maneuver and walking. La- angioplasty was performed [4]. Thus, it was suspected boratory data showed slight anemia (hemoglobin 11.5 g/ that hemodynamic failure contributed to the pathophysi- day), hypoalbuminemia (3.8 g/dL, normal range 3.8– ology. That is, under the chronic low output condition 5.3 g/dL), and mild renal dysfunction (serum creatinine of uncontrolled HOCM, factors such as occasional dehy- (Cre) 1.33 mg/dL, normal range 0.6–1.2 mg/dL; blood dration and bradycardia had reduced systemic perfusion, urea nitrogen (BUN) 39 mg/dL, normal range 9–23 mg/ especially that of intestinal blood flow. We performed a dL; estimated glomerular filtration rate (eGFR) 39.5 mL/ pacemaker implantation with right ventricular apical minute/1.73 m2), and no signs of dyslipidemia or dia- pacing to improve our patient’s hemodynamic condition. betes mellitus (low density lipoprotein cholesterol After dual-chamber pacemaker implantation, pacing AV 98 mg/dL; glycosylated hemoglobin 5.3 %; fasting blood delay was optimized for a minimum LVOT pressure gra- glucose 100 mg/dL). Our patient’s activated partial dient and mitral valve regurgitation under echocardio- thromboplastin time (33.1 seconds) and prothrombin graphic estimations (AV delay 100 milliseconds for time (13.0 seconds) were within the normal ranges. Gas- LVOT 14.6 mmHg, Fig. 4). Following these procedures, troscopy and colonoscopy examinations detected no ab- our patient’s appetite and daily activity improved dra- normal findings, while a computed tomography (CT) matically. In addition, his blood pressure improved sig- scan with contrast enhancement demonstrated moderate nificantly, his renal function became normal (Cre, stenosis of the celiac artery and severe stenosis of the in- 0.83 mg/dL; BUN, 25 mg/dL; eGFR, 66.2 mL/minute/ ferior mesenteric artery (Fig. 1). In addition, significant 1.73 m2), and a chest X-ray examination showed signifi- dilatation of the bowels was observed by CT and chest cant improvement of intestinal dilatation (Fig. 2), sug- X-ray examinations (Fig. 2). gesting increase in systemic and intestinal perfusion. Fig. 1 Enhanced computed tomography showed severe atherosclerosis of the arteries. The aorta showed broad calcifications, and the celiac artery showed moderate stenosis, although the lumen of the SMA was relatively patent. In addition, stenosis of the IMA was very severe. IMA inferior mesenteric artery, SMA superior mesenteric artery Hamaoka et al. Journal of Medical Case Reports (2016) 10:271 Page 3 of 5 artery and disturb blood flow (median arcuate ligament syndrome) [5, 6]. Intestinal circulation consists of an abundant collateral blood supply, and chronic intestinal ischemia is associated with high-grade stenosis or occlusion of two or more of the three major vessels: the celiac artery, the superior and inferior mesenteric arter- ies [7, 8]. In our case, the arterial lesions were relatively mild compared with previous reports [9, 10]; thus, we hypothesized that our patient’s major symptoms were not due to his arterial lesions alone. Specifically, our patient’s symptoms depended greatly on his hemodynamic condition, which caused a flow dis- crepancy between demand and supply [1]. Once an ab- Fig. 2 Chest X-ray examinations pre-PMI demonstrated significant dominal attack occurred, our patient’s blunted oral dilatation of the bowels; however, this dilatation was improved after PMI with right ventricular apical pacing. PMI